[Transient inhibitors developed in two cases of hemophilia A].
This report describes the transient development of inhibitors in two cases of hemophilia A. Case 1 was an 8-month-old boy who suffered from subcutaneous hematoma with extremely low level (1.5%) of plasma factor VIII activity (F VIII:C). He showed an inhibitor titer of 2.5 Bethesda units (BU)/ml when he suffered from measles at age 24 months with poor clinical response to factor VIII. This inhibitor elevated to 11.6 BU/ml at 29 months, but completely disappeared at 32 months. Case 2 was a 5-month-old boy who suffered from subcutaneous hematoma with extremely low level (< 1%) of plasma F VIII:C. At 8 months his inhibitor titer peaked at 19 BU/ml but subsequently declined to 11.5 BU/ml at 12 months and completely disappeared at 14 months. A review of the literature revealed that the median (range) titer of transient inhibitor initially developed at 2 (0.5-19) BU/ml on 225 (79-448) days, immediately peaked at 3.6 (0.5-60) BU/ml and disappeared by 665 (282-1140) days, suggesting that careful monitoring of F VIII inhibitors is required in the treatment of hemophilia A.
Fujiwara
F
F
Department of Pediatrics, Nantan General Hospital.
Kanoh
G
G
Ogawa
H
H
Takaya
K
K
Hibi
S
S
Shima
M
M
Yoshioka
A
A
Imashuku
S
S
D001323:Autoantibodies / Q000032:analysis; D005169:Factor VIII / Q000008:administration & dosage / Q000037:antagonists & inhibitors* / Q000276:immunology; D006467:Hemophilia A / Q000097:blood* / Q000276:immunology; D006801:Humans; D007223:Infant; D008297:Male
D001323:Autoantibodies; D005169:Factor VIII